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\begin{document}

\title{Multimodality imaging of multiple giant right coronary artery aneurysms
combined with anomalous aortic origin of left coronary artery}



\author[1]{Mingxing Xie}%
\author[1]{Yu Cai}%
\author[1]{Shenglei Shu}%
\author[1]{Yali Yang}%
\affil[1]{Wuhan Union Hospital}%


\vspace{-1em}



  
  \date{May 25, 2024}


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\selectlanguage{english}
\begin{abstract}
We report a rare case of multiple giant coronary artery aneurysms
combined with anomalous aortic origin of left coronary artery in a
30-years old man precisely diagnosed by multimodality imaging, including
echocardiography, coronary computed tomographic angiography (CCTA),
cardiac magnetic resonance imaging (CMR) and selective coronary
angiography. The imaging results were finally confirmed by surgery. We
present the clinical value of multimodality imaging in diagnosing
coronary artery aneurysm and anomalous origin.%
\end{abstract}%



\sloppy


\textbf{Introduction}

Coronary artery aneurysm(CAA)is defined as a local dilatation in the
coronary artery which is 1.5 times greater than the adjacent normal
segment, with the prevalence of 0.3-5\% in patients undergoing coronary
angiography\textsuperscript{{[}1{]}}. The giant CAA is defined as CAAs
over 20mm with an incidence of 0.02\% to 0.2\% in cardiac surgical
population \textsuperscript{{[}2{]}}. Anomalous aortic origin of a
coronary artery (AAOCA) is a rare congenital abnormality of the origin
or course of a coronary artery that arises from the aorta with the
prevalence of 0.1\%-1\% in both the adult and pediatric
populations\textsuperscript{{[}3{]}}. However, the optimal treatment of
CAA and AAOCA based on detailed information of coronary
arteries\textsuperscript{{[}1, 3{]}}. Multimodality imaging, including
echocardiography, coronary artery angiography, coronary computed
tomographic angiography (CCTA), cardiac magnetic resonance imaging
(CMR), has been suggested in diagnosing CAA and
AAOCA\textsuperscript{{[}4{]}}. We report a rare case of multiple giant
CAAs combined with anomalous aortic origin of left coronary
artery,presenting the vital role of multimodality imaging in diagnosing
and developing optimal individualized treatment strategies.

\textbf{Case report}

A 30-years old man was presented with exertional chest pain repeatedly,
without hypertension and dyslipidemia. On physical examination, his
blood pressure was 105/55mmHg, heart rate is 70 bpm, cardiac murmur was
not obvious. Electrocardiogram showed the sign of ischemia in inferior
wall and previous infarction in interventricular~septum. Hypersensitive
troponin I level was 37.5 ng/L, the blood NT-ProBNP level was 376.0
pg/ml, the inflammatory markers were normal. Transthoracic
echocardiography(TTE) found an enlarged mass (5.1\selectlanguage{ngerman}×4.3cm) close to right
coronary artery opening, with thrombus contained probably and no flow
signal inside, compressing the anterior~part~of~tricuspid~annular and
right ventricle without abnormal hemodynamics (Figure 1A and B). Besides
of that, the decreased motion of middle segment anterior septal wall~was
detected by TTE. Coronary angiography showed that there were multiple
aneurysms in proximal and middle part of right coronary artery (RCA),
but distal part of RCA can't be displayed (Figure 2A). The left anterior
descending (LAD) and left circumflex artery (LCX) were occluded in
proximal part, and collateral circulation was developed around (Figure
2B). To clearly displaying the distal part of RCA, CCTA was performed
and found a string of aneurysms distributed in distal part of RCA
(Figure 3A). Unexpectedly, CCTA found the origin of
left~coronary~artery~was abnormal, which arising from left side of
sinotubular junction and part of left main coronary arteries
distributing between the ascending aorta and the main pulmonary artery
(Figure 3B). CMR found some of aneurysms had thrombose inside (Figure
3C), and distinctly displayed the course of left main coronary arteries
distributing between two arteries (Figure 3D). Taken imaging
characteristics of coronary artery into consideration, the diagnosis
mostly be suspected as multiple giant right coronary artery aneurysms
with thrombus combined with anomalous aortic origin of left coronary
artery. Finally, coronary artery bypass graft surgery was performed, and
intraoperative finding demonstrated the suspected diagnosis. In the
intraoperative view, there were two giant coronary artery aneurysms with
thrombus adjacent to each other in proximal and mid part of right
coronary artery (Figure 4).

\textbf{Discussion}

The pathogeny of coronary artery aneurysm includes atherosclerosis,
Kawasaki disease, inflammation, trauma, infection, connective tissue
disease and so on\textsuperscript{{[}5{]}}. Even though atherosclerosis
is the most common cause of CAA in adult\textsuperscript{{[}5{]}}, the
patient in this case has no evidence of atherosclerosis. The cause of
CAA in this patient may mostly be Kawasaki disease or congenital
abnormality, with the evidence of the multiple coronary artery aneurysms
and the negative serology investigations.

Anomalous origin of the coronary artery (AOCA) associated with sudden
cardiac death and myocardial ischemia\textsuperscript{{[}3{]}}. The
presented hypotheses about sudden cardiac death were based on anatomic
and physiologic properties of the anomalous coronary, while the
triggering factors of the myocardial ischemia may be the insufficient
coronary reserve\textsuperscript{{[}3{]}}.

The CAA and AOCA, both of the coronary anomalies are asymptomatic
usually, just abnormal electrocardiogram was shown\textsuperscript{{[}2,
3{]}}. In this case, the patient has chest pain symptom and abnormal
electrocardiogram. Multimodality imaging plays a vital role in
developing optimal management by revealing giant CAAs combined with
anomalous aortic origin of left coronary artery, but also providing
detailed information of coronary artery, myocardial wall contraction and
the cardiac hemodynamics. Coronary angiography is useful in evaluating
stenosis degree of coronary artery and compensative collateral
circulation, while the overlook of CAAs and its relationship with
adjacent constitution was not clear. CTA displayed multiple CAA
scattered throughout the right coronary artery and anomalous origin of
the left coronary artery arises from sinotubular junction. In delayed
enhancement sequence, CMR detects the thrombus contained in some
coronary artery aneurysms. Transthoracic echocardiography provides
information about the change of cardiac hemodynamics, abnormal motion of
ventricular wall, and the effects of giant CAAs on cardiac structure and
function.

Based on the anatomic subtypes of AAOCA and characteristics of giant
CAAs, it is necessary to develop individualized treatment plan based on
the detailed information of coronary artery. In this case, multimodality
imaging indicated that the patient had intervention indication. Then,
coronary artery bypass graft surgery was performed, and he recovered
uneventfully and no major adverse cardiac events were reported during
the 6-months follow-up.

\textbf{Sources of Funding:}

This work was not supported by any foundation.

\textbf{Conflict of interest:}

The authors declare that they have no conflict of interest.

\textbf{Consent:}

Informed consent was obtained from the patient for the publication of
this article.

\textbf{Ethical approval:}

This study was approved by the Ethics Committee of Tongji Medical
College, Huazhong University of Science and Technology.

\textbf{References}

{[}1{]} Kawsara A, Nunez G I, Alqahtani F, et al. Management of Coronary
Artery Aneurysms{[}J{]}. JACC Cardiovasc Interv, 2018,11(13):1211-1223.

{[}2{]} Crawley P D, Mahlow W J, Huntsinger D R, et al. Giant coronary
artery aneurysms: review and update{[}J{]}. Tex Heart Inst J,
2014,41(6):603-608.

{[}3{]} Brothers J A, Frommelt M A, Jaquiss R, et al. Expert consensus
guidelines: Anomalous aortic origin of a coronary artery{[}J{]}. J
Thorac Cardiovasc Surg, 2017,153(6):1440-1457.

{[}4{]} Grani C, Buechel R R, Kaufmann P A, et al. Multimodality Imaging
in Individuals With Anomalous Coronary Arteries{[}J{]}. JACC Cardiovasc
Imaging, 2017,10(4):471-481.

{[}5{]} Abou S S, Ozden T O, Taskoylu O, et al. Coronary Artery
Aneurysms: A Review of the Epidemiology, Pathophysiology, Diagnosis, and
Treatment{[}J{]}. Front Cardiovasc Med, 2017,4:24.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image1/image1}

\end{center}
\end{figure}

\textbf{Figure 1.} Transthoracic echocardiography found that the giant
aneurysm (yellow arrows) close to right coronary artery opening (A),
with thrombus contained and no flow signal inside, compressed the
anterior~part~of~tricuspid~annular and right ventricle, but the
diastolic~flow velocity~at~tricuspid~valve~orifice and middle part of
right ventricular~cavity was normal (B).

(LA:left atrium; LV: left ventricle; RA: right atrium; RV: right
ventricle; RVOT: right ventricular outflow tract; AO: aorta)\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image2/image2}

\end{center}
\end{figure}

\textbf{Figure 2.} Coronary angiography presented two giant coronary
artery aneurysms (yellow arrows) in proximal and middle part of RCA (A).
The proximal part of LAD and LCA were nearly occluded and plenty of
collateral circulation was around (B).\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image3/image3}

\end{center}
\end{figure}

Figure 3. Coronary computed tomographic angiography showed two adjacent
giant CAAs (yellow arrows) in proximal and middle part of RCA, and
multiple CAAs in the distal part (yellow rectangle) (A). The left
coronary artery (red arrow) originated from aortic sinotubular junction
(B). Cardiac magnetic resonance imaging found thrombus (yellow asterisk)
in those CAAs (C), and part of left main trunk (red arrow) distributing
between the aorta and the main pulmonary artery (D).

(LA:left atrium; LV: left ventricle; RV: right ventricle; AO: aorta; PA:
pulmonary artery)\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image4/image4}

\end{center}
\end{figure}

Figure 4. Intraoperative view of two giant right coronary artery
aneurysm (yellow dotted line).

(AO: aorta; PA: pulmonary artery)\selectlanguage{english}
\begin{figure}[H]
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\includegraphics[width=0.70\columnwidth]{figures/Figure-1/Figure-1}

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\includegraphics[width=0.70\columnwidth]{figures/Figure-2/Figure-2}

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\includegraphics[width=0.70\columnwidth]{figures/Figure-3/Figure-3}

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\end{figure}\selectlanguage{english}
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\includegraphics[width=0.70\columnwidth]{figures/Figure-4/Figure-4}

\end{center}
\end{figure}

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